Finck Camille, Gambron Tersia, Benchimol Lionel, Camby Severine, Morsomme Dominique
ENT Department, University Hospital of Liege, Belgium.
Faculty of Medecine, Uliege, Belgium.
Heliyon. 2023 Oct 11;9(10):e20869. doi: 10.1016/j.heliyon.2023.e20869. eCollection 2023 Oct.
Neuroborreliosis is part of advanced stage of Lyme disease and often characterized by damage to the cranial and/or peripheral nerves. Involvement of one or both recurrent nerves is rare. Diagnosis is often difficult and based on a set of clinical manifestations, biological arguments, and cerebrospinal fluid (CSF) analysis. A 70-year-old man was referred to our Voice Clinic with a 3-month history of dysphonia caused by right vocal fold paralysis (VFP) without any cutaneous symptoms of tick bite or erythema migrans in the previous weeks and normal initial radiological examination (neck and thorax CT). Methylprednisolone had already been prescribed but without any clinical improvement. Late biological investigation 3 months after initial symptoms of VFP showed high IgG (93 U/mL; reference <10 U/mL) against (BB), which was confirmed by two immunoblot markers (VIsE, p39 antigens). Therefore, a possible manifestation of Lyme disease with involvement of the right inferior laryngeal nerve was suspected, namely Lyme neuroborreliosis. However, given the spontaneous recovery of the patient after 7 months without any adapted antimicrobial regimen treatment, the diagnosis of neuroborreliosis was not confirmed by a lumbar puncture. Nineteen months later, the patient presented again for the same symptomatology but as left VFP. High IgG (68 U/mL) and IgM (>6, reference <0.90) levels against BB were confirmed by immunoblot. Subsequently, lumbar puncture was performed and revealed IgG against BB at 46.1 UA/mL (reference<5.5 UA/mL) in the CSF, with an extremely high IgG intrathecal synthesis antibody index (281.33, positive if > 1.5). Intrathecal antibody synthesis is the gold standard for Lyme neuroborreliosis demonstrating a specific immune response to BB in the central nervous system, but with the limitation of persistence for years after eradication. Our patient did not exhibit pleocytosis in the CSF. Therefore, two criteria of the European Federation of Neurological Societies (EFNS) guidelines are fulfilled for possible neuroborreliosis. Doxycycline treatment led to rapid recovery in less than 8 weeks and normal mobility of the left vocal fold. Because of this very uncommon clinical presentation with two successive episodes of VFP for no other obvious reason and serological evidence from the serum and CSF during the second episode, we consider it possible that the first episode of VFP could also have been a manifestation of neuroborreliosis. This case is the first report of possible relapse of laryngeal palsy successively on the right, and then the left side as a manifestation of Lyme neuroborreliosis.
神经莱姆病是莱姆病晚期的一部分，常表现为颅神经和/或周围神经受损。一侧或双侧喉返神经受累的情况较为罕见。诊断往往困难，需依据一系列临床表现、生物学证据及脑脊液（CSF）分析。一名70岁男性因右侧声带麻痹（VFP）导致声音嘶哑3个月被转诊至我们的嗓音诊所，此前数周无蜱叮咬或游走性红斑的皮肤症状，初始放射学检查（颈部和胸部CT）正常。患者已接受甲泼尼龙治疗，但无临床改善。VFP初始症状出现3个月后的后期生物学检查显示，抗伯氏疏螺旋体（BB）的IgG水平较高（93 U/mL；参考值<10 U/mL），通过两种免疫印迹标志物（VIsE、p39抗原）得以证实。因此，怀疑为莱姆病累及右侧喉下神经的一种可能表现，即莱姆神经疏螺旋体病。然而，鉴于患者在未接受任何针对性抗菌治疗的情况下7个月后自发康复，腰椎穿刺未确诊神经疏螺旋体病。19个月后，患者因相同症状再次就诊，此次为左侧VFP。免疫印迹证实抗BB的IgG（68 U/mL）和IgM（>6，参考值<0.90）水平升高。随后进行腰椎穿刺，脑脊液中抗BB的IgG为46.1 UA/mL（参考值<5.5 UA/mL），鞘内IgG合成抗体指数极高（281.33，>1.5为阳性）。鞘内抗体合成是莱姆神经疏螺旋体病的金标准，表明中枢神经系统对BB有特异性免疫反应，但在根除后可持续数年。我们的患者脑脊液中无细胞增多现象。因此，符合欧洲神经科学会联合会（EFNS）指南中可能的神经疏螺旋体病的两条标准。强力霉素治疗在不到8周内使患者迅速康复，左侧声带活动恢复正常。鉴于这种非常罕见的临床表现，即无其他明显原因先后出现两次VFP发作，且第二次发作时有血清和脑脊液的血清学证据，我们认为第一次VFP发作也可能是神经疏螺旋体病的一种表现。该病例是首例关于莱姆神经疏螺旋体病表现为先后右侧和左侧喉麻痹可能复发的报告。
